Primary ovarian leiomyoma: A case report

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InternationalJournalofSurgeryCaseReports5(2014)665–668

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International

Journal

of

Surgery

Case

Reports

j o u r n al ho m e p a g e :w w w . c a s e r e p o r t s . c o m

Primary

ovarian

leiomyoma:

A

case

report

Mine

Islimye

Taskin

_,

_Emine

_Ozturk

_,

_Fatma

_Yildirim

_,

Necmettin

Ozdemir

_,

_Umit

_Inceboz

a_{BalıkesirUniversitySchoolofMedicine,DepartmentofObstetricsandGynecology,Turkey} b_{EgeUniversitySchoolofMedicine,DepartmentofPathology,Turkey}

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Articlehistory: Received4July2014 Accepted24July2014 Availableonline4August2014

Keywords: Ovary Leiomyoma

Immunohistochemistry

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INTRODUCTION:Primaryovarianleiomyomaisararebenigntumouroftheovaryseeninwomenbetween

20and65yearsold.Itisusuallydiagnosedincidentallyduringpelvicexaminationorpathologic

exami-nationaftersurgery.

PRESENTATION OF CASE: Wedescribe a case of unilateral, ovarianleiomyoma. Transvaginal

ultra-sonography and magnetic resonance imaging (MRI) revealed a right adnexial mass. Unilateral

salpingo-oophorectomywasperformed, andhistologicalexaminationrevealedaleiomyomaarising

primarilyintheovary.Thediagnosiswasconfirmedimmunohistochemically.

DISCUSSION:Thetumourmaybeasymptomaticormaymanifestwithlowerabdominalpainlikein

ourcase.Thedefinitivediagnosisoftheselesionsisdifficultpriortosurgicalremoval.Becausethere

isnopathognomonicsymptomsorcharacteristicimagingfindings.Thecorrectdiagnosisofanovarian

leiomyomarequiresidentificationofthesmoothmusclenatureofthetumour.

CONCLUSION:Thisraretumouroftheovaryshouldbeconsideredinthedifferentialdiagnosisofsolid

ovarianmasses.Animmunohistochemicalanalysisisrecommendedfordefinitivediagnosis.

©2014TheAuthors.PublishedbyElsevierLtd.onbehalfofSurgicalAssociatesLtd.Thisisanopen

accessarticleundertheCCBY-NC-SAlicense(http://creativecommons.org/licenses/by-nc-sa/3.0/).

1. Introduction

Leiomyomaisoneoftherarestsolidtumoursoftheovary;it accountsfor0.5–1%ofallthebenignovariantumours.1 Approx-imately 70 cases have been reported in the literature.Ovarian leiomyomasareparticularlyunilateralandsmall,andtheymost commonly occurin women aged 20–65. The majorityof these tumours are discovered incidentally, with about 80% of the casesoccurringin premenopausalwomen.2 _{Patientsareusually} asymptomatic,andthetumourismostcommonlydiagnosed unin-tentionallybyhistologicalexaminationofovariantissueafteran ovariectomyforsolidovarianmass.Ovarianleiomyomasprobably arisefromsmoothmusclecellsintheovarianhilarbloodvessels, butotherpossibleoriginsarecellsintheovarianligament,smooth musclecellsormultipotentialcellsintheovarianstroma, undif-ferentiatedgermcells,andcorticalsmoothmusclemetaplasia.3_In thisstudy,wereportacaseofaprimaryovarianleiomyomaina 42-year-oldwoman.

∗ Correspondingauthorat:BalikesirUniversitySchoolofMedicine,Department ofObstetricsandGynecology,10145C¸a˘gıs¸Kampüsü,Balıkesir,Turkey.

Tel.:+902666121454/1289;fax:+902666121294.

E-mailaddresses:minetaskin1302@yahoo.com.tr,minetaskin@balikesir.edu.tr

(M.I.Taskin).

2. Casereport

A42-year-oldgravida1para 0woman wasadmitted toour university hospital (Balıkesir, Turkey) with a history of lower abdominalpainanddesireforpregnancy.Herhistorywas unre-markablewiththeexceptionofthreeinvitrofertilisationcycles for male-factor infertility and one pregnancy that was aborted in the sixth weekof gestation. Physicaland abdominal exami-nationfindings werenormal.On vaginal examination,a mobile massapproximately4cmin diameterwasdetectedintheright lower abdomen. Her CA 125, CA15-3, CA 19-9, CEA,and AFP valueswerewithinnormallimits.Transvaginalultrasonography revealedarightadnexialmass38.9mm×36mmindiameterthat showed a homogeneously isoechoic pattern (Fig. 1). The mass wassolidandwellcircumscribed,andpathologicbloodflowwas not detectedin Doppler ultrasonography.Pelvic MRI showeda 4cm×3cm,well-circumscribedsolidtumourin therightadnex thatexhibitedlow-intensitysignalsonbothT1-andT2-weighted images.Alaparatomywasperformedundergeneralanaesthesia. Duringlaparotomy,inspectionoftheuterusandadnexesrevealed asolid,firm,oval,right-sidedovariantumourwithasmooth sur-face,approximately4cmindiameter.Thetumourwasdistinctly separatedfromtheuterusandexhibitednoadhesiontoor infil-trationofthesurroundingstructures.Therewasnoaccompanying uterinemass.Theleftsalpinx,ovarywerenormaloninspectionand

http://dx.doi.org/10.1016/j.ijscr.2014.07.020

2210-2612/© 2014The Authors.Published byElsevier Ltd.on behalf ofSurgicalAssociates Ltd. Thisis an openaccess articleunder theCC BY-NC-SA license

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666 M.I.Taskinetal./InternationalJournalofSurgeryCaseReports5(2014)665–668

Fig.1.Rightovariansolidmass.

wereleftintact.Rightunilateralsalpingo-oophorectomywas per-formed,andfrozensectioningrevealedabenigntumoursuggesting thecomaorfibroma.Aftersurgery,histologicaland immunhisto-chemicalexaminationresultedinadiagnosisofprimaryovarian leiomyoma.Pathologicexaminationrevealedatumourcomposed ofinterlacingbundlesoffusiformcells,resemblingaleiomyoma (Fig.2).Therewasnoatypiaorpleomorphism,andmitoticcount andnecrosiswereabsent(Fig.3).Immunohistochemicalstaining showedstrongand diffusepositive staining forsmooth muscle actin(SMA)(Fig.4).Ki-67proliferationindexwaslow(Fig.5).The postoperativeperiodwasuneventful.

3. Discussion

Primaryleiomyomaoftheovaryisaveryrarebenigntumour, usuallydetectedincidentallyduringroutinepelvicexamination, during surgery, or after surgical removal of the ovary. Most leiomyomas of the ovary are small, usually less than 3cm in diameter.6_{Mostofthepatientsareasymptomaticor,asinourcase,}

Fig.2. Fasciclesofsmoothmusclecells(H&E,×100).

haveonlycomplaintsoflowerabdominalpain.Incontrast,giant ovarianleiomyomascanbepresented withascite,hydrothorax, hydronephrosis,orslightlyelevatedlevelsoftumourmarker,CA 125.1,4,5

Casereportshavedemonstratedapredominanceofunilateral leiomyomas,butbilateralovarianleiomyomashavebeenreported inpaediatricandyoungadultpatients.Bilateralcaseshavenotbeen reportedinpatientsovertheageof35.7_{Inlinewiththeliterature,} ourpatientwas42yearsoldandhadunilateralovarian leiomy-oma.Ovarianleiomyomasoftencoexistwithuterineleiomyomas, butsometimestheycanbeasecondaryorigin.Thatis,subserosal pedunculateduterineleiomyomacanloseitsattachmenttothe uterusandconnecttotheovary.Thishadobviouslynotoccurredin thepresentcase,becausetheuteruswasnormalanddidnotexhibit anysignsofleiomyoma.

Fig.3. Thesmoothmusclecellsareuniformlyspindleshapedorelongatedwith blunt-endedorcigar-shapednuclei.Mitoticactivityisabsentorverylow,and cel-lularandnuclearpleomorphismsareabsent.

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Fig.4. Markedimmunoreactivityforsmoothmuscleactin(×200).

Fig.5.VerylowKi-67proliferationindex(×400).

Theliterature hasreportedthatmany patientswithovarian leiomyomasarenulligravidas.Thissuggeststhat oestrogenmay playa roleinthedevelopmentofovarianleiomyomas.Another possiblemechanismsuggeststhattumourmayarisein develop-mentallyabnormalovaries.8_{Inourcase,normalovariantissuewas} presenthistologically.Thissuggeststhatthetumourcanoriginate fromthesmoothmusclecellsinthewallsofbloodvessels,inthe corticalstroma,inthehilus,inthecorpusluteum,orintheovarian ligament.

Inthepreoperativeperiod,differentialdiagnosiscanbe diffi-cultfromovarianthecomaand/orfibromasoftheovary.Because ultrasonographyvisualisesovarianleiomyomasasintrapelvicsolid tumours,isoechoicwiththemyometrium,itisdifficultto distin-guishthemfrompedunculateduterineleiomyomasandothersolid ovariantumours.MRIisoften ausefuladjunctto ultrasonogra-phyfor thepurpose ofdiagnosing indistinct pelvic masses.9 _In thepresentcase,MRIrevealedawell-circumscribedsolidtumour inthepelvisthatdemonstratedlow-intensitysignalsonT1-and T2-weightedimages.Whensuchasolidovariantumourwiththe similarfindings of uterineleiomyoma detectedin MRI,ovarian leiomyomacanbeconsidered.

Thecorrectdiagnosisofanovarianleiomyomarequires identi-ficationofthesmoothmusclenatureofthetumour.Inthepresent case, an immunohistochemical analysis confirmed our diagno-sis,althoughfrozensectioningrevealedfibrothecoma.Thediffuse strongpositive stainingforSMA ischaracteristic ofleiomyoma. Thecoma couldalsobeconsideredin differentialdiagnosis,but it doesnotexpressSMA.1,3,6_{Ovarian leiomyomasmust alsobe} differentiated from leiomyosarcomas.For this aim,pathologists usecriteriasuchasmitoticcount,cytologicalatypia,andtumour necrosis.3 _{In ourcase, noneofthesecriteria weredetected.} Ki-67isanindexproteinthataffectsgrowthcontrolinleiomyoma monoclonalcellsandishelpfulintheevaluationofcharacteristics ofuterineleiomyomaneoplasticprocesses.10_{Inthepresentcase,} theKi-67proliferationindexwasassessedandfoundtobevery low.

Acommonsurgicalapproachtoovarianleiomyomasin middle-aged to elderly patients is hysterectomy in conjunction with bilateral salpingo-oophorectomy. For bilateral ovarian leiomy-omas,bilateraloophorectomyisoftenrequired.Weietal.reported acaseinwhichpedunculatedunilateralovarianleiomyomaand ovarianpreservationwerepossible.7_{Inthepresentcase,we} per-formed unilateral salpingo-oophorectomy. Because the tumour wasindistinguishablefromtheovary,thetumourwasalso uni-lateralandbenign.

4. Conclusion

The present study presents a rare case of primary ovarian leiomyoma.Preoperativediagnosiscanbedifficultwithsolid ovar-iantumours,animmunohistochemicalanalysisisrecommended fordefinitivediagnosis.

Conflictofinterest

Authorsdeclarethatthereisnoconflictofinterest. Funding

None. Ethicalapproval

Writteninformedconsentwasobtainedfromthepatientfor publicationofthiscasereportandaccompanyingimages

Authorscontribution

Mine Islimye Taskinand UmitInceboz contributed to study designandwriting.EmineOzturkcontributedtodatacollection. Necmettin Ozdemirand Fatma Yılmazcontributed tohistologic analysis.

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